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27

Facial Characterization of Infants With Cleft Lip and Palate Using a

Three-Dimensional Capture Technique

C.A. HOOD, M.SC.D., F.D.S., R.C.P.S., B.D.S.

M.T. HOSEY, D.D.S. , M.SC. (MED SCI), F.D.S., R.C.P.S., B.D.S.

M. BOCK, PH.D., B.SC., B.MATH.(HONS)

J. WHITE, M.ORTH., F.D.S., R.C.P.S., B.D.S.

A. RAY, M.B.B.S., M.S., F.R.C.S.(PLAST)

A.F. AYOUB, PH.D., M.D.S., F.D.S., R.C.P.S., F.D.S.R.C.S., B.D.S.

Objective: To characterize the soft tissue features of infants with unilateral

cleft lip (UCL) and unilateral complete cleft lip and palate (UCLP) prior to pri-

mary surgery and compare with noncleft controls.

Design: Prospective controlled capture of the facial morphology of infants

using a noninvasive three-dimensional stereophotogrammetry method.

Participants: 23 children with presurgical cleft: 11 UCL (M 6, F 5); 12

UCLP (M 9, F 3), and 21 noncleft controls (M 7, F 14) were imaged at

approximately 3 months of age (range 10 to 16 weeks).

Main Outcome Measure: Accurate, repeatable quantification of facial soft tis-

sues in infants with clefts prior to surgery.Results: Significant differences (p .05) were found between the UCLP

group and UCL and control groups in anatomical and soft nose width, cleft-

side alar wing length, and nasal tip horizontal displacement. Both cleft groups

were significantly different from controls and from each other in cleft-side nos-

tril dimensions, alar wing angulation, columella angle, and alar base to corner

of mouth dimension; alar base width; and soft tissue defect in nose and the

lip and philtrum length bordering the cleft. Significant differences between

clefts and controls were identified in the nostril and philtrum on the noncleft

side.

Conclusions: The use of children with UCL as controls for UCLP studies is

inappropriate. This technique overcame the limitations of direct measurement

of infant faces to aid the surgeon in the planning and subsequent re-evaluation

of surgical rationale.

KEY WORDS: children, cleft lip, cleft lip and palate, facial morphology, infants,

presurgical, soft tissues, 3D, unilateral

The goals of surgical intervention for the repair of cleft lip

and palate are the restoration of normal morphology and func-

tion, without disruption of growth potential. However, the im-

pact of primary cleft surgery on facial growth is a subject of

continued debate. Attempts to unravel the compounding ef-

Dr. Hood is a Clinical Lecturer and Dr. Hosey is a Senior Clinical Lecturer/ Honorary Consultant in Pediatric Dentistry, Dr. Bock is a Lecturer, Department

of Statistics, Mr. White is a Clinical Lecturer in Orthodontics, and Professor

Ayoub is a Senior Clinical Lecturer/Honorary Consultant in Oral and Maxil-

lofacial Surgery and Head of Biotechnology and Craniofacial Research Group,

University of Glasgow, Glasgow, Scotland. Mr. Ray is a Consultant in Plastic

Surgery, Canniesburn Hospital, Glasgow.

This project has been funded by a grant from The Chief Scientist Office in

Scotland and The National Lottery Charities Board through CLAPA, UK. Grant

K/OPR/2/2/O366.

Submitted November 2002; Accepted February 2003.

Address correspondence to: Dr. Ashraf F. Ayoub, Glasgow Dental Hospital

and School, 378 Sauchiehall Street, Glasgow G2 3JZ, United Kingdom. E-mail

[email protected].

fects of tissue hypoplasia associated with the cleft defect itself,

and the consequences of surgery have been hindered by a lack

of objective means to quantify facial parameters in infancy. A

paucity of comparative normative data, and the perception that

early assessment of surgical outcome is not possible, further

complicates the picture.

Cephalometric studies have provided valuable information

about hard tissues and limited soft tissue information in youngchildren with clefts (Dahl et al., 1982; Hermann et al., 1999).

The use of cephalometric radiography, particularly in the

young patient, has been questioned (Shaw et al., 1992a, 1992b;

Mackay et al., 1994). Examination of the soft tissue profile of

individuals with orofacial clefts may indicate the extent of

maxillary hypoplasia and provide more significant information

about suture growth patterns than examination of hard tissues

(Shaw et al., 1992b). However, examination of soft tissue pro-

file provides only limited understanding of the relationships of

structures in the midline and cannot quantify aberrant anatom-

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28 Cleft Palate–Craniofacial Journal, January 2004, Vol. 41 No. 1

ical position or asymmetry (Molsted et al., 1992). A full ap-

preciation of the complex deficiencies and 3-dimensional (3D)

nature of the cleft malformation is a prerequisite for quanti-

fying the magnitude of the anomaly and measuring change

following surgical repair.

Facial soft tissue characteristics of infants with untreated

cleft lip and cleft lip and palate have been reported by variousresearchers, employing a diversity of data collection methods

(Bacher et al., 1998; Fisher and Mann, 1998; Fisher et al.,

1999; Hermann et al., 1999; Hurwitz et al., 1999; Yamada et

al., 1999). With the exception of direct anthropometry studies

(Farkas, 1994; Mulliken et al., 2001), few have compared their

findings to a matched control group. When planning such in-

vestigations, quantitative examination methods require com-

parison with appropriate population norms (Farkas, 1997).

Moreover, the influence of racial or ethnic diversity on mor-

phometric findings should not be underestimated (Hajnis et al.,

1994).

Whatever method is adopted, the capture of an image of a

3-month-old infant remains a challenge. Despite considerableadvances in laser-scanning techniques (O’Grady and Antony-

shyn, 1999; Duffy et al., 2000), the time required for image

acquisition (10 to 13 seconds) precludes their use in young

children. Researchers have resorted to image capture or direct

measurement of facial morphology under general anesthesia,

immediately prior to operative procedures, despite the likeli-

hood of facial distortion by the presence of an endotracheal

tube (Hurwitz et al., 1999; Mulliken et al., 2001). Infant se-

dation has been necessary to allow collection of certain an-

thropometric measurements using calipers, particularly around

the eyes and nose (Farkas et al., 1993). Facial alginate im-

pressions, a technique so potentially hazardous as to require

an emergency intubation kit and the presence of a neonatolo-

gist, has also been advocated as a suitable data-gathering meth-

od in infants (Bacher et al, 1998). Although these various tech-

niques are noninvasive with respect to exposure to x-rays, they

require pharmacological intervention together with additional

support staff and facilities, which renders them less useful for

long-term monitoring of surgical outcome (Poswillo, 1990;

American Academy of Pediatric Dentistry 1999; Cote et al.,

2000).

The three-dimensional stereophotogrammetry (C3D) system

has been developed to overcome these difficulties. The system

allows noninvasive capture of 3D photorealistic facial mor-

phology in 50 milliseconds without the need for sedation. This

allows collection of serial records for monitoring treatment,

and comparing progressive outcomes in a practical, user-

friendly way. The development, clinical application, and re-

producibility of this technique have been previously reported

(Ayoub et al., 1997, 1998, 2003; Al-Omari et al., 2003; John-

son et al., 2003).

The aims of this investigation were to characterize the facial

soft tissue features in infants with unilateral cleft lip (UCL)

and complete unilateral cleft lip and palate (UCLP), prior to

cheiloplasty, and compare these soft tissue findings with a peer

group control.

METHOD

Ethical approval was granted from North Glasgow Univer-

sity Hospitals NHS Trust and Royal Hospital for Sick Children

NHS Trust local ethics committees. There are comprehensive

mechanisms for the recording and documentation of all cleft

births in Scotland (Scottish Congenital Anomalies Register atthe Information and Statistics Division of the Common Ser-

vices Agency).

Identification and recruitment of children with cleft was un-

dertaken in consultation with National Managed Clinical Net-

work for Cleft Services in Scotland (CLEFTsis).

Twenty-three children with unilateral clefts, 11 (six boys,

five girls) with UCL and 12 (nine boys, three girls) with UCLP,

had 3D images captured prior to surgical intervention, at ap-

proximately 3 months of age (range 10 to 16 weeks). 21 age-

matched infant controls (7 boys, 14 girls) were recruited

through maternity units and baby clinics in Glasgow.

The C3D system configured for clinical facial imaging con-

sists of two pods, each with three cameras. Two monochromecameras serve to form a stereo baseline and are synchronized

to capture images illuminated by special texture flash projec-

tors. A third color camera captures the natural photographic

appearance of the subject under normal white-light flash.

The system was calibrated before and after each capture

session. A calibration target of accurately known dimensions

was imaged in a variety of target poses, built, and attached to

the sets of captured images. This contains the necessary in-

formation required to create a depth map. Several sets of facial

images were captured in the same session and those with facial

expression at rest (minimal muscle activity) selected and built

at low resolution (0.005 voxel size). Built 3D range data were

examined for smoothness, integrity (Fig. 1C) and evidence of

intrinsic or extrinsic movement (such as blinking, changes in

facial expression during image capture). The desired models

were then edited to remove extraneous background material

and built at high resolution (0.002 voxel size). The final part

of the selection process involved examination of the color tex-

ture map, and the best quality model for each individual was

then selected for analysis. This final C3D output can be viewed

as a solid wire frame or shaded model (Fig. 1A through 1C).

Infants were photographed at rest, on a parent’s lap, looking

slightly upward toward the operator (Fig. 2). 3D models were

built and anthropometric landmarks (Table 1, Fig. 3) identified

on each using the Facial Analysis Tool, custom software that

allows model manipulation and analysis on a computer screen.

To facilitate direct comparison, models of children with right-

sided clefts were reflected so that every cleft appeared on the

left side. Models were orientated to a standard position, and

two operators (C.A.H., J.W.) systematically identified highly

reproducible sets of 35 landmarks (average placement error 0.5

mm). Operators were trained in placement of anthropometric

landmarks and calibrated (intraoperator error 0.5 mm, inter-

operator error 0.81 mm). Selection of landmarks on screen

generates a set of x, y, and z coordinates, which describe the

spatial orientation of facial features. Linear distances angles

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Hood et al., 3D PRESURGICAL FACIAL CHARACTERIZATION 29

FIGURE 1. Output of three-dimensional stereophotogrammetry (C3D) system. A. Photorealistic solid C3D model. B. Wire frame. C. Shaded C3D model

illustrating smooth range data.

FIGURE 2. Three-dimensional stereophotogrammetry camera set-up.

Child is imaged, unrestrained, on a parent’s lap. Capture time is 50 mil-

liseconds.

and ratios defining key facial parameters were generated (Table

2) using the Facial Analysis software.

One-way analysis of variance statistical tests were per-

formed to compare mean individual measurements in all three

groups ( p .05). Tukey’s multiple comparison test calculated

confidence intervals for each possible pair of groups.

RESULTS

In an effort to report on the findings in this study in a clin-

ically meaningful way, the results are grouped according to

facial region. These are presented in terms of the cleft defor-

mity (i.e., UCL and UCLP) and then compared with the con-

trol group. The ear landmarks were difficult to identify in some

models and excluded from this analysis. The complete expo-

sition of the results is shown in Table 3.

Upper Face

Compared with controls, intercanthal width was significant-

ly increased in the UCLP group but not the UCL group. On

the cleft side, the distance from nasion to endocanthion was

longer in UCL and UCLP, compared with controls. Although

this was more marked in the UCLP group, the difference be-

tween cleft groups was not significant. There were no differ-

ences in ocular and biocular widths between cleft groups and

controls.

Nostril Dimensions

In both cleft groups, the cleft-side nostril was significantly

increased in all dimensions, compared with controls. There

were highly significant differences in the noncleft side nostril,

compared with controls. Nostril long axis and nostril floor

widths were increased in UCL and UCLP, relative to controls,

but there were no significant differences between the cleft

groups.

Nose: Horizontal Dimensions

Both cleft groups had significantly increased alar base

widths, compared with controls. This dimension was also sig-

nificantly wider in the UCLP group than the UCL group.

Soft nose width (al-al) and anatomic nose width (ac-ac)

were increased in the UCLP group but not the UCL group.

There were no differences in soft nose width: mouth width

ratios between groups but, as expected, an increased anatomic

nose base width; mouth width ratio was detected in the UCLP

group.

The size of the soft tissue defect in the nose is gauged by

the difference in cleft and noncleft side nostril floor widths.

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TABLE 1 Landmark Definitions*

Region Landmark Definition

Eyes

Eyes

Eyes

Eyes

Forehead

1

2

3

4

5

enL

enR

exL

exR

n

Endocanthion, left

Endocanthion, right

Exocanthion, left

Exocanthion, right

Nasion

NoseNose

Nose

Nose

Nose

67

8

9

10

prnsn

sn0R

sn0L

cR

PronasaleSubnasale

Edge of the collumellar base right

Edge of the collumellar base left

Highest point of the collumella (reflected onto nostril) right

Nose

Nose

Nose

Nose

Nose

11

12

13

14

15

cL

sbalL

sbalR

acL

acR

Highest point of the collumella (reflected onto nostril) left

Subalare left

Subalare right

Alar left

Alar right

Nose

Nose

Nose

Nose

Nose

16

17

18

19

20

alL

alR

al0oL

al0oR

al0iL

Alare left

Alare right

Midpoint on outer margin of left ala, opposite al0iL

Midpoint on outer margin of right ala, opposite al0iR

Midpoint on outer margin of nostril left

Nose

Lips

LipsLips

Lips

21

22

2324

25

al0iR

chR

chLcphR

Is

Midpoint on outer margin of nostril right

Cheilion right

Cheilion leftCrista philtri right

Labiale superioris

Lips

Lips

Lips

Lips

Lips

26

27

27

28

29

cph0R

cphL

cphL

stoi

li

Crista philtri landmark surrogate on major segment, bordering cleft (left-sided cleft)

Crista philtri landmark surrogate on minor segment, cleft side (left-sided cleft)

Crista philtri left, controls only

Stomion inferioris

Labiale inferioris

Lips

Other

Other

Other

Other

Other

30

31

32

33

34

35

sl

pg

obsR

obsL

obiR

obiL

Sublabialis

Pogonion

Otobasion superior right

Otobasion inferior left

Otobasion superior right

Otobasion superioris left

* Farkas, 1990, 1994; Hurwitz et al., 1999; Duffy et al., 2000.

FIGURE 3. Photokey showing positions of anthropometric landmarks.

On average, there was a 2-mm discrepancy in the UCL group,

rising to 10 mm in the UCLP group.

Nose: Vertical Dimensions

In the UCL group, there were no differences in nose dorsum

length or nasal tip base (sn-prn), in comparison with controls.

In the UCLP group, nose dorsum length was longer than in

both the controls and UCL group.

As with the UCL group, there was no difference in nasal

tip–base distance in the UCLP group; however, nasal tip an-

gulation was increased in both cleft groups, highlighting flat-

tening and skewing of the nasal tip in the preoperative face.

Lateral nose dorsum lengths were similar in clefts and controls.

Alar Wing

The length of the alar wing on the cleft side was increased

only in the UCLP group. The UCL group had normal alar

lengths on both the cleft and noncleft sides.

Alar wing angulation was more acute on the noncleft side,

with a corresponding obtuse angle on the cleft side in the

UCLP group. However, this feature was not found in the UCL

group, reflecting the lesser degree of nasal tip displacement

and alar wing flattening in these children.

Columella

The width and height of the columella were normal in the

cleft groups. The columella angle on the cleft side, however,

was more obtuse in both cleft groups. There was no corre-

sponding decrease in columella angle on the noncleft side.

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TABLE 2 Facial Measurements, Ratios, and Angle Definitions*

Measurement Definition

Upper face

Biocular width

Ocular width

Intercanthal width

Endocanthion to nasion

ex-ex

ex-en

en-en

en-n

Nostril Dimensions

Nostril floor width

Nostril long axis 1

Nostril width 2

sbal to sn0

sbal-c

sn0-al0i

Nose horizontal dimensions

Alar base width

Anatomic nose width (alar base facial insertion width)

Soft nose width

Anatomic nose base/mouth width ratio

Soft nose/mouth width ratio

Size of soft tissue defect in the nose difference between L&R nasil floor widths

Nasal tip horizontal displacement (angle)

sbal-sbal

ac-ac

al-al

ac-ac:ch-ch

al-al:ch-ch

(sbalL-sn-0L)-(sbalR-sn0R)

acR-prn-acL

Nose vertical dimensions

Nose dorsum length

Lateral nasal length

Nasal tip-base

Nasal tip angulation

n-prn

en-ac

sn-prn

n-prn-sn

Alar wing

Projective alar length

Alar wing angulation (R&L)

ac-prn

ac-prn-sn

Columella

Columella height

Columella thickness

Columella angulation (R&L)

sn0-c

sn0-sn0

sbal-c-sn0

Mouth

Lower lip length

Lower lip vermillion height

Mouth width

stoi-sl

stoi-li

ch-ch

Philtrum

Size of defect in midportion of upper lip difference in paramedial vertical upper lip heights on noncleftand cleft sides

Size of cleft in upper lip distance between philtral points

Medial height of upper lip skin

Philtral point to ipsilateral corner of mouth

Philtral point to ipsilateral alar base

Alar base to ipsilateral corner of the mouth

(sn0-cph0)-(sn0-cph)

cph-cph

sn-ls

cph-ch (noncleft side) chp-ch (cleft side)

cph-sbal (noncleft side) cph-sbal (cleft side)

sbal-ch

Others

Nasolabial angle

Protrusion of Is relative to sn

Upper face height

Face height

prn-sn-Is

Is-n-sn

n-sn

n-pg

Mouth

Mouth width was unaffected by the presence of a cleft.

Compared with controls, cph-ch (corner of mouth to high point

Cupid’s bow) was shorter on cleft side in UCL but not on the

noncleft side. In contrast, in UCLP this distance was shorter

on both the cleft and noncleft sides.

There were no differences in upper lip lengths on the cleft

side between controls and UCLP or UCL groups. However, on

the noncleft side, this dimension was significantly shorter in

the UCL group.

Philtrum

The paramedial philtrum length on the noncleft side was sig-

nificantly shorter in the UCL group, compared with controls.

There were no significant differences seen in the UCLP group,

however. On the opposite side of the philtrum, bordering the cleft,

both the UCL and UCLP groups showed significant shortening

of the opposite side of the philtrum.

Comparison of the paramedial philtrum dimensions, on the

noncleft side and bordering the cleft, gave an indication of the

quantitative defect in the midportion of the upper lip. In the

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TABLE 3 Facial Measurements: UCL, UCLP, and Control Groups at 3 Months†

Measurements

Mean (mm)

Control UCL UCLP

ANOVA

Value

Tukey’s Confidence Interval

UCL:Control UCLP:Control UCL:UCLP

Upper face

exL-exR

exL-enL

exR-enRenL-enR

enL-n

enR-n

65.75

20.39

20.2726.71

14.70

15.51

65.65

20.43

20.1926.77

16.42

15.93

67.12

20.25

19.9128.42

17.53

15.68

.429

.932

.801

.043*

.000***

.772

2.88/ 0.56

3.4/ 0.02

3.96/ 1.70

Nostril dimensions

sbalL-sn0L

sbalR-sn0R

sbalL-cL

sbalR-cR

sn0L-al0iL

sn0R-al0iR

4.54

4.20

6.08

5.81

4.39

4.75

7.19

5.05

7.84

6.99

6.09

5.18

16.20

5.53

15.33

7.74

9.65

5.05

.000***

.000***

.000***

.000***

.000***

.486

4.44/ 0.85

1.55/ 0.15

3.4/ 0.12

1.99/ 0.37

2.55/ 0.83

13.41/ 9.92

2.01/0.65

10.84/ 7.65

2.71/ 1.14

6.09/ 4.42

11.03/ 7.0

9.32/ 5.65

4.53/ 2.59

Nose horizontal dimensions

sbalL-sbalR

acL-acR

alL-alR

acL-acR:chL-chR

alL-alR:chL-chR(sbalL-sn0L)-(sbalR-sn0R)

10.80

24.85

22.44

0.87

0.780.25

15.21

25.14

20.30

0.88

0.702.14

23.77

30.45

25.12

0.97

0.8010.68

.000***

.000***

.002**

.000***

0.71.000***

6.63/ 2.2

3.66/ 0.14

15.12/ 10.82

7.41/ 3.78

5.3/ 0.06

0.18/ 0.04

12.14/ 8.72

11.04/ 6.07

7.4/ 3.21

7.83/ 1.8

0.18/ 0.02

10.51/ 6.56

Nose vertical dimensions

n-prn

enL-acL

enR-acR

sn-prn

n-prn-sn

19.59

19.42

19.45

9.85

111.18

21.15

19.43

18.94

9.28

118.42

21.55

20.56

20.51

9.14

122.15

.015*

.165

.080

.100

.000*** 12.14/ 2.33

3.69/ 0.25

15.74/ 6.2

Alar wing

acL-prn

acR-prn

(acR-prn)-(acL-prn)

acR-prn-sn

acL-prn-sn

acR-prn-acL

16.13

16.80

0.67

53.81

53.99

98.30

16.82

16.38

0.44

46.32

60.29

98.32

21.23

16.80

4.43

43.43

74.51

105.72

.000***

.660

.000***

.000***

.000***

.001**

1.82/13.16

11.14/ 1.46

6.72/ 3.49

3.49/6.71

4.85/15.89

25.22/ 15.81

12.17/ 2.68

6.27/ 2.54

2.13/5.85

19.64/ 8.79

12.87/ 1.93

Collumella

sn0L-cLsn0R-cR

sn0R-sn0L

sbalL-cL-sn0L

sbalR-cR-sn0R

2.222.35

5.13

37.84

36.41

1.852.61

4.93

62.16

32.59

1.982.76

4.68

114.49

29.63

.312

.382

.354

.000***

.449

43.53/ 9.94 90.55/ 57.87 66.15/ 28.8

Mouth

chR-chL

cphR-chR

cphL-chL

28.91

17.90

17.78

28.78

16.62

14.01

31.39

15.68

11.61

.052

.025*

.000*** 1.0/6.55

0.3/4.17

3.47/8.87

Lower lip

sl-stoi

li-stoi

10.86

5.62

9.97

4.04

10.28

4.23

.090

.000*** 0.75/2.39 0.59/2.18

Philtrum

cphR-sn0R

cphR-sbalR

cphL-sn0L vs cph0R-sn0L

ls-sn

sbalL-chL

11.99

10.41

11.97

10.76

22.65

9.26

8.13

6.32

6.91

20.07

11.16

10.19

4.60

6.95

17.57

.003**

.020*

.000***

.000***

.000***

0.94/4.53

0.33/4.22

4.14/7.16

2.43/5.27

0.6/4.57

5.9/8.84

2.42/5.18

3.16/7.02

0.02/3.42

0.28/4.73

sbalR-chR

cphL-cphR vs cphL-cphR

cph-ls vs cph0R-ls

cphL-sbalL

cphR-ls

23.08

6.38

3.31

9.93

3.37

20.07

15.28

2.79

11.17

2.84

20.44

19.95

3.03

8.33

3.70

.000***

.000***

.090

.111

.217

1.04/4.97

11.72/ 6.09

0.73/4.55

16.31/ 10.83 7.83/ 1.51

Other

prn-sn-ls

ls-n-sn

n-pg

n-sn

138.74

2.64

58.08

24.88

137.50

2.31

58.80

26.83

136.21

2.92

58.82

27.53

.716

.658

.879

.001** 3.75/ 0.16 4.4/ 0.91

† VCL unilateral cleft lip; UCLP complete unilateral cleft lip and palate; ANOVA analysis of variance.

* p .05; ** p .01; *** p .001.

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UCL group, the mean was 2.94 mm on average, and in the

UCLP group, the mean was 6.56 mm.

Medial philtrum length (sn-ls) was significantly shorter in

both UCL and UCLP, regardless of severity of cleft.

In both cleft groups, the distance from the alar base to the

corner of the mouth on the cleft and noncleft sides was shorter,

compared with controls. Furthermore, on the cleft side, thisdistance was significantly shorter in the UCLP group, com-

pared with the UCL group.

The average size of the cleft in the upper lip was greater in

the UCLP group, 19.95 mm, compared with 12.46 mm in the

UCL group.

Lower Lip

There were no significant differences among the three

groups in respect of total lower lip length. However, lower

vermillion length was significantly shorter in both cleft groups,

compared with controls.

Other Features

Nasolabial angles in the cleft groups were similar to con-

trols. There were no significant differences in upper lip prom-

inence between cleft groups and controls, measured by the

position of the center of Cupid’s bow relative to the columella

base.

Upper face height (n-sn) was increased in the cleft groups.

Face height, measured from nasion to pogonion (n-pg), was

similar in both the cleft and control groups.

DISCUSSION

Dahl (1970) described the differences found in UCL as mild

and limited to the cleft region. Other hard tissue studies of

children with clefts have supported the use of the child with

UCL as a ‘‘control’’ (Hermann et al., 1999). This approach

cannot be adopted for the study of soft tissues because of the

heterogeneity of clefts affecting the lip and primary palate. Our

study found significant differences among the UCL, UCLP,

and control groups in a number of soft tissue parameters.

Where the UCL group differed from controls, this was also

expressed as a significant difference in the UCLP group, com-

pared with controls, with one notable exception: philtrum di-

mensions on the noncleft side.

In general, significant differences between the UCL and

UCLP groups occurred in soft tissue parameters in which the

UCL group was more like controls (anatomical and soft nose

width, cleft-side alar wing length, and nasal tip horizontal dis-

placement). In addition, significant differences were found in

which cleft groups differed not only from controls but also

from each other (cleft-side nostril dimensions, alar wing an-

gulation, columella angle, alar base to corner of mouth dimen-

sion, alar base width; soft tissue defect in the nose and lip,

philtrum cleft border).

Comparison of other cleft groups with a UCL ‘‘control’’

group will cloud the analysis of the effects of surgical inter-

ventions on facial growth. The findings in this study reinforce

the need for peer group comparison with matched children

without cleft.

Scotland has a particular predilection for congenital malfor-

mations, and approximately 100 new cases of orofacial clefting

occur each year. In common with Northern Ireland, there is aremarkably high ratio (almost 1:1) of cleft lip and cleft lip and

palate to isolated cleft palate in the Scottish population (Wom-

ersley and Stone, 1987; FitzPatrick et al., 1994; Gregg et al,

1994; Stone and Dolk, 1994). This is not typical of the United

Kingdom or other cleft centers, generally, in which the ratio

is 2:1. Farkas (1997) recommended that ethnically and racially

differing regions should develop their own control population

norms for statistical analysis. This is the first report to char-

acterize the facial soft tissues of Caucasian infants with cleft

lip and cleft lip and palate in the Scottish population, prior to

primary repair.

Although the children were matched for age, there were

more girls than boys in the control group. The possibility of sexual dimorphism was not explored. However, a recent study

by Yamada (Yamada et al., 2002a) demonstrated few sex dif-

ferences in the facial soft tissues in infants. Moreover, the se-

verity of the cleft deformity may ‘‘mask’’ sex differences pre-

operatively, as has been reported in hard tissue studies (Krog-

man et al., 1982). Much larger sample sizes will be required

to consider this further.

Increased intercanthal width has been previously described

in studies of children with cleft lip (Dahl et al., 1982; Friede

et al., 1986). In this study, increased intercanthal width was a

feature of the UCLP group but not the UCL group. Biocular

width (exR-exL) and right and left ocular widths were of nor-

mal proportion. The increased intercanthal width in the UCLP

group could be explained by an apparent increased distance

from inner canthus to nasion on the cleft side. This dimension

was also significantly increased in the UCL group, which sug-

gests subtle variation in anteroposterior position of the eye

between cleft types. Linear analysis alone cannot explain this

fully, and a more comprehensive 3D analysis is warranted.

The increase in the upper face height in both cleft groups

in this study is in agreement with that reported by other re-

searchers (Farkas, 1990).

Although our study did not examine individual landmark

positions, our finding of increased nasal floor width on the

noncleft side confirms that the noncleft subalare (sbal) is dis-

placed in relation to the columellar base. Indeed, Fisher et al.

(1999) noted that this point was farther from the midline than

that on the cleft side and that there was a marked deviation of

the columellar base, subnasale (sn), from the midline. Given

that the surgeon uses the noncleft side as a frame of reference

to correct asymmetry, increased nostril dimensions could have

implications for residual nasal deformity, particularly in func-

tion. This may require consideration at the time of cleft repair.

During cleft surgery, the alar base is an important clinical

dimension to correct. The width of the alar base of the nose

(sbal-sbal) in UCLP was more than twice that found in con-

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trols. In the UCL group, this dimension was also increased,

but the alar base facial insertion (ac-ac) was similar to controls.

These findings can be explained by the more lateral and in-

ferior position of the subalare point on the cleft side and ever-

sion of the alar base. This is in response to the torque effect

produced by contraction of facial muscles that have lost their

medial insertion, but deeper soft tissue remains anchored toperiosteum (Malek, 2001). An exaggeration of this effect is

seen in UCLP and compounded by flattening and elongation

of the ala on the cleft side, evident in the increased anatomical

and soft nose widths and shortening of the distance from the

corner of mouth to the alar base on the cleft side.

Proportion ratios of nose base and mouth widths have been

advocated in the assessment of surgical outcome and residual

deformity (Vegter et al., 1997). Farkas et al. (1993) proposed

that anatomical nose width was better determined between ac-

ac, as opposed to al-al, because this is really a measure of soft

nose width. In our study we chose to measure both dimensions

and then calculate proportion indices (Table 2). We found that

mouth widths were similar in both cleft groups and controls.Even though both al-al and ac-ac were significantly increased

in the UCLP group, the nose base: mouth width ratio did not

reflect this, when the al-al:ch-ch was used as a basis for the

proportion calculation. In contrast, a proportion index based

on ac-ac:ch-ch showed significant differences between the

UCLP group and controls. The use of soft nose width (al-al)

in calculations of nose:mouth width ratios may render the mea-

sure too insensitive for the detection of subtle morphological

differences in young children.

Both cleft groups’ collumellae were displaced but of normal

height and width. A more obtuse angle was found on the cleft

side in both the cleft groups, but there was no corresponding

decrease in columellar angle on the noncleft side. The displace-

ment of the noncleft subalare point may be masking this feature.

In infants with UCLP, nose dorsum dimensions, cleft-side

alar wing dimensions, and nasal tip angulation characterize the

flattening and horizontal skewing of the nasal tip in one di-

rection and the columella base in the other. In common with

other studies (Hurwitz et al., 1999; Duffy et al., 2000; Yamada

et al., 2002a), the point chosen to represent the most prominent

anterior point of the nose (prn) does not necessarily coincide

with the anatomical tip. Inherent difficulties in the identifica-

tion and reproducibility of the anatomical tip necessitate the

selection of a close surrogate.

Few researchers have managed to quantify nostril shape in

children. Using this morphometric approach, we were able to

map important nostril parameters in young children with cleft

lip and palate prior to surgical intervention.

Hermann et al. (1999) reported a more prominent upper lip

in UCLP measured in profile from cephalometric radiographs.

This feature was not found in this study. In children with clefts,

unopposed cleft orbicularis oris fibers together with major

maxillary segment rotational displacement combined to pro-

duce unilateral shortening of the upper lip. This becomes even

more striking because of the tilt or inclination of Cupid’s bow

(Fisher and Mann, 1998; Fisher et al., 1999; Hermann et al.,

1999; Hurwitz et al., 1999; Yamada et al., 1999). The present

study showed that short philtrum and upper lip dimensions

were a feature of the UCL group on the noncleft as well as

on the cleft side. In contrast, the UCLP group had normal

philtrum dimensions on the noncleft side, although signifi-

cantly decreased distances bordering the cleft. We can postu-

late that this is the result of outward rotational muscular pullon the lip soft tissues in UCL, whereas in UCLP there is com-

plete bone separation of the segments, with concomitant major

segment displacement; the net effect is less tension on the lip

soft tissues on the noncleft side.

Mouth widths in infants with cleft were similar to noncleft

controls. This is an important baseline to establish because

mouth width is a useful outcome measure following surgery.

Indeed, significantly narrower mouth widths have been re-

ported in studies of older children and adults with cleft (Farkas

and Lindsay, 1973; Zhu et al., 1994)

The width of the lower lip was significantly shorter in both

cleft groups, compared with controls. All children in this study

were photographed at rest, with a lip-apart posture. Anthro-pometric measurements relating to lower lip and vertical

mouth dimensions should not rely on the stomion landmark

(sto), which must be identified with the lips together. It is well

documented that young children with clefts are obligate mouth

breathers (Hairfield et al., 1988), and a posture of ‘‘lips to-

gether at rest’’ is not achievable. Many of the authors who use

an anthropometric landmark-based approach fail to acknowl-

edge this point (Hurwitz et al., 1999; Mishima et al., 2002;

Yamada et al., 2002a, 2002b). In view of this, the stomion

inferior (stoi) landmark was selected for our analysis. Stomion

superior (stos) does not exist in the preoperative cleft face,

but, in practical terms, measuring lower vermillion height

helps estimation of upper vermillion height, which has impli-

cations for the design of surgical approach.

A quantitative assessment of facial asymmetry should be

included in any evaluation of craniofacial anomaly. This is an

area for future study (Hood et al., 2002). Nevertheless, the

indirect C3D anthropometry technique can overcome many of

the limitations of direct measurement of infant faces, with the

added benefit of a 3D coordinate–based analysis. Accurate,

repeatable soft tissue measurement can be used to quantify

characteristics of the cleft condition in advance of surgery and

so aid the surgeon in the planning and subsequent re-evalua-

tion of surgical rationale.

CONCLUSIONS

This study demonstrated the use of a noninvasive C3D ap-

proach to facilitate the objective comparison of cleft defects.

Significant differences were identified between infants with

unilateral clefts of the lip and primary palate and UCLP and

age-matched noncleft peer group controls.

The development of appropriate local baselines to establish

the normal facial character within any given population should

be seen as a prerequisite for comparison of surgical outcomes.

The C3D system provides a possible solution to the problem

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of standardization of methods of interpretation of findings be-

fore surgery and during follow-up. This will facilitate objec-

tive comparison of results of different surgical techniques and

timings of cleft repair in the future.

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